Human Reproduction, Vol. 17, No. 5, 1363-1366,
May 2002
© 2002 European Society of Human Reproduction and Embryology
Heterotopic triplet pregnancy: report of a case with bilateral tubal pregnancy and an intrauterine pregnancy: Case report
Department of Obstetrics and Gynecology, Shin Kong Wu Ho-Su Memorial Hospital, No. 95, Wen Chang Road, Shih Lin District, Taipei, Taiwan
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Abstract |
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The first report of an ectopic pregnancy following IVF was published in 1976, and since then heterotopic pregnancies (HPs) have been reported at an increasing rate. Although cases of the co-existence of a bilateral tubal and an intrauterine pregnancy following IVF–embryo transfer have been reported, a case of heterotopic triplet pregnancy caused by unilateral tubal embryo transfer has not yet been published in the literature. Here we report on a 38-year-old women (gravida 3, para 1) with a history of infertility who presented to our infertility clinic for evaluation. Hysterosalpingography revealed bilaterally patent Fallopian tubes and stricture of the cervical canal. She conceived after receiving HMG combined with pure FSH, followed by IVF–tubal embryo transfer. Four embryos were replaced into the right tube. Approximately 5 weeks after tubal embryo transfer, the patient presented with lower abdominal tenderness and shock due to internal bleeding. She underwent an emergency laparotomy under the impression of HP. Bilateral tubal pregnancy with right tubal rupture was noted during the operation. The post-operative course was uneventful. Early intervention and thorough inspection of the peritoneal cavity in patients with haemodynamic instability can prevent jeopardizing the life of the mother as well as the ongoing pregnancy.
Key words: bilateral tubal pregnancy/heterotopic pregnancy/IVF-tubal embryo transfer/triplet pregnancy
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Introduction |
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The first IVF cycle that resulted in an ectopic pregnancy was reported by Steptoe and Edwards (Steptoe and Edwards, 1976
). Since then, heterotopic pregnancies have increased alongside the advent of assisted reproductive technology (ART) (KlipStein and Oskowitz, 2000). More than a hundred heterotopic pregnancies (HPs) following ART have been reported during the last decade (Rojansky and Schenker, 1996). However, only three cases of bilateral tubal HP were noted, which in two cases occurred following IVF–embryo transfer (Hanf et al., 1990; Jonler et al., 1995) and one after gamete intra-Fallopian transfer (GIFT) (Wang et al., 1996).
Several risk factors predispose the patient to HP after GIFT. These factors include endometriosis, peritubal adhesion and an excessive number of oocytes transferred (Molloy et al., 1990; Li et al., 1992). In the present case, the possible aetiologic factors, variation in clinical presentation and outcome of management are discussed.
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Case report |
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A 38-year-old woman (gravida 3, para 1) with a history of infertility presented to our infertility clinic for evaluation. She underwent laparoscopic surgery for minimal endometriosis and mild pelvic adhesion 5 years prior to admission. She had previously undergone infertility treatment with artificial insemination of husband (AIH) and one pregnancy was achieved; two dilatation and curettage were done due to a blighted ovum, and one failed. Upon examination, HSG revealed bilaterally patent Fallopian tubes, cervical stricture and probable uterine synechiae near the left cornual area. Ovarian stimulation utilizing HMG combined with pure FSH therapy was started. Ovarian response was monitored using vaginal ultrasound. HCG 5000 IU was administered 36 h prior to ultrasound-directed oocyte recovery. A total of 18 oocytes was collected and nine fertilized.
We performed tubal embryo transfer because of cervical stenosis from previous conization ~6 months earlier. Four embryos (two grade II embryos, i.e. regular blastomeres and minor fragments, one grade III embryo, i.e. irregular blastomere, and one grade IV embryo, i.e. regular or irregular blastomeres and many fragments) were replaced into the right Fallopian tube. The luteal phase was supported by using HCG. Two weeks later, the patient's ß-HCG was 366 mIU/ml. Twenty-eight days after tubal embryo transfer, a live intrauterine pregnancy was detected under transvaginal ultrasonography.
Approximately 5 weeks after tubal embryo transfer, the patient came to our emergency room with a complaint of cramping lower abdominal pain, nausea, tenesmus and vaginal bleeding. Her blood pressure was 82/50 mmHg and pulse 92 beats/min. Physical examination revealed diffuse abdominal and cervical motion tenderness. Her haemoglobin was 7.9 gm/dl, white cell count 31 500/ul and a pregnancy test was positive. Transvaginal sonography showed bilateral multicystic ovaries with accumulation of fluid in cul-de-sac and Morrison's pouch (Figures 1 and 2). A positive fetal heart beat in utero also confirmed an intrauterine pregnancy. She was admitted for an emergency operation under the impression of a possible corpus luteum cyst rupture with persistence of bleeding or a heterotopic pregnancy with hypovolemic shock. Laparotomy revealed a ruptured right tubal pregnancy with haemoperitoneum. Upon inspection, the left tube was dilated resembling the right tube.
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After informing the families, we decided to perform bilateral salpingectomy because it was impossible to preserve both tubes. At least 500 ml of peritoneal blood was evacuated and 4 units of packed red blood cells were transfused to correct anaemia. The post-operative course was uneventful, and the patient was discharged in a stable condition. The pathologic examination confirmed degenerated chorionic villi in the right tube and vascularized chorionic villi in the left. Pregnancy continued with no further complications and the patient delivered vaginally a healthy boy at term.
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Discussion |
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Spontaneous HP is a rare event, occurring in one in 30000 pregnancies (De voe and Pratt, 1948
). As more and more infertile couples turn to ART to achieve a successful pregnancy, the incidence of HP has expectedly increased to 1.2 and 2.9% in two different large studies (Dimitry et al., 1990; Dor et al., 1991).
Recently, the incidence of HP after GIFT has been reported to be 0.83% (Li et al., 1992). Meanwhile, the proportion of HP occurring after tubal embryo transfer has been estimated to be ~0.5% of all pregnancies (Wu et al., 1995). Hence, the predisposing factors of ART-related HP should be recognized to allow prompt management. Techniques of embryo and gamete transfer, number and quality of embryos and gametes replaced, pelvic and tubal condition, hormonal milieu and superfecundations are well known risk factors (Yovich et al., 1985; Molloy et al., 1990; Li et al., 1992; Wang, 1996). Excessive medium and improper catheter insertion may lead to dispersion of embryos by the so-called `spray and drift' effect (Molloy et al., 1990; Marcus et al., 1995; Klipstein and Oskowitz, 2000). Furthermore, it is logical to assume that deliberately placing up to four embryos in one Fallopian tube will increase the chance of ectopic pregnancy occurring simply from the increased genetic material in the tube (Molloy et al., 1990; Rojansky and Schenker, 1996). The alteration of the local progesterone/estradiol ratio by clomiphene citrate may disturb oviductal peristasis (Marcus et al., 1995). Early rising of progesterone would favour opening of the isthmus and myorelaxation of the tube (Salat-Baroux et al., 1985).
There are several reports in the literature suggesting that GnRH analogue use may be linked to a higher rate of ectopic pregnancy in the IVF population (Klipstein and Oskowitz, 2000). If the patient has previous pelvic inflammatory disease, there will be an obvious increase in ectopic pregnancies (Marcus et al., 1995). Sometimes, minor subclinical tubal pathology may also be a risk factor (Li et al., 1992; Mechiers et al., 1992). It is now recommended that the number of transferred embryos should not exceed three, especially in women with history of tubal pathology. However, several ART centres have reported success after GIFT in case of endometriosis and even peritubal adhesion (Novy et al., 1991). GIFT has recently been extended to patients with tubal pathology, thus again increasing the risk of extrauterine implantation (Strowitzki et al., 1993).
In this case, the patient had unilateral minimal endometriosis and pelvic adhesion, a high estradiol level (1580 pg/ml) on the day of HCG injection, and transfer of four embryos in one tube, all of which are considered risk factors of HP. Therefore, no more than three embryos should ever be transferred in order to decrease risk, as well as multiple pregnancies. Instead of IVF–embryo transfer, we performed tubal embryo transfer on a patient with existing risk factors because we encountered difficulty in inserting the Wallace catheter into the uterine cavity. This experience demonstrates the importance of observing rule of the risk for preventing adverse events.
The most important aid in the diagnosis of heterotopic pregnancy is the utilization of a high-resolution transvaginal ultrasonography (Guirgis and Craft, 1991; Louis-Sylvestre et al., 1997). In high risk patients, especially those whom conceived via ART, Guirgis and Craft recommended a routine ultrasound scanning for ectopic or heterotopic pregnancy at 4–6 weeks after transfer of embryos (Guirgis and Craft, 1991). Some authors may suggest that sonography has low sensitivity of 0.56 (Ankum et al., 1993). In fact, a pre-operative diagnosis of HP was present in only 10% of cases (Fernandez et al., 1993). Hence, Mol et al. suggested that the use of probabilistic decision rules in the algorithms for the work-up of suspected ectopic pregnancy increases the diagnostic performance of flexible algorithms as compared with inflexible algorithms using rigid cut-off values (Mol et al., 1999a). On the other hand, abdominal pain, rebound tenderness on abdominal examination, fluid in the pouch of Douglas at transvaginal sonography examination and a low serum haemoglobin concentration were independent predictors of tubal rupture and/or active bleeding (Mol et al., 1999b). In our case, bilateral ovaries were markedly enlarged after ovulation induction, possibly masking the ectopic pregnancy. A significant amount of peritoneal fluid was thought to be ascites resulting from ovarian hyperstimulation syndrome. It may be difficult to distinguish ascites from haemoperitoneum under ultrasound. The finding of an ectopic pregnancy upon laparotomy confirmed our initial impression.
The management of heterotopic pregnancy still remains controversial. Several authors have mentioned the value of a laparoscope in the diagnosis and treatment; its safety is also well documented (Dietz et al., 1993; Wang et al., 1998; Ludwig et al., 1999). The success of laparotomy is also mentioned in several studies (Fujii et al., 1996; Barnett et al., 1998; Andersen, 1999). Operative management is still a mainstay, but it involves surgical and anaesthetic risk to both the mother and fetus. Although it was reported that laparotomy did not seem to interrupt intrauterine pregnancy, others have reported up to 40% loss of viable fetuses after surgery (Oehninger et al., 1988).
So far, there are only three reports of bilateral tubal with intrauterine pregnancy after ART (two cases achieved by embryo transfer, one by GIFT). This is, to our knowledge, the first report where both rare patterns of implantation abnormality occur simultaneously after tubal embryo transfer. One possible explanation is that one embryo from the four that were transferred subsequently migrated from the site of placement, crossed the uterine cavity and implanted in the lumen of the contralateral tube, as has been postulated by Klipstein et al. (Klipstein and Oskowitz, 2000 ). In the previous reports of bilateral tubal pregnancy, the contralateral tubal pregnancy was diagnosed days to weeks after the initial surgery (Sherman et al.,1991). In our case, the contralateral tubal pregnancy was noted incidentally. Therefore, if HP is suspected prior to operation, thorough inspection of the abdomen, pelvis and contralateral tube is mandatory, and we suggest that no more than three embryos should be transferred during the IVF–tubal embryo transfer procedure.
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1 To whom correspondence should be addressed. E-mail: M004407{at}ms.skh.org.tw
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Submitted on February 22, 2001; resubmitted on November 26, 2001; accepted on December 10, 2001.
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